Abstract
Le Fort I osteotomy is the technique most applied worldwide in the treatment of dentoskeletal deformity involving the maxilla. Even though it is considered a very safe technique with good intra- and postoperative results, many complications have been described. This paper presents a case of carotid jugular fistula developed in a 22-year-old white male submitted to Le Fort I osteotomy for the treatment of anteroposterior maxillary deficiency, and discusses the possible aetiology and management of this serious complication.
The Le Fort I osteotomy, generally considered a safe procedure, is one of the techniques most commonly applied worldwide to correct deformities affecting the middle third of the face, and is also useful in cases that require access to skull base tumours. However, complications have been reported in the literature, the most common of which are haemorrhage, infection, and airway obstruction. Other complications have also been reported, such as perioperative dental trauma, exaggerated maxillary impaction with an unfavourable aesthetic result, oroantral and oronasal fistulae, nerve or duct damage, and, with lower incidence, velopharyngeal dysfunction and vascular complications.
A traumatic arteriovenous fistula is an abnormal communication between an artery and a vein caused by an incomplete tearing of both vessels. Most cases of traumatic arteriovenous fistulae involve the internal carotid artery and are more frequent after blunt or penetrating trauma, and more rarely as a result of surgical procedures such as orthognathic surgery .
Lesions to the internal carotid artery during Le Fort I osteotomy have been described in the literature. Although rare, it is a serious complication .
The aetiology of carotid jugular fistulae related to the treatment of dentoskeletal deformities is controversial, some authors attributing this complication to the sharp bony edge of the pterygoid complex detached during surgical fracture and others to the anatomical characteristics of the patient .
The following case report describes the occurrence of a right carotid–jugular arteriovenous fistula after a Le Fort I osteotomy performed for maxillary advancement and intrusion and discusses the possible causes and preventive measures to avoid this serious kind of complication.
Case report
A 22-year-old white male diagnosed with anteroposterior maxillary deficiency was admitted to the hospital for an orthognathic surgery procedure that consisted of a 5-mm maxillary advancement and a 2-mm intrusion. The usual Le Fort I osteotomies were performed, applying the reciprocating saw at the anterior and lateral aspect of the maxilla, the specific chisel at the nasal septum, and a curved chisel between the pterygoid process and the maxillary tuberosity. Nonetheless, downfracture of the maxilla was unsuccessful at the first attempt; all osteotomy cuts were then re-examined, and the same instruments and additional pressure (that is still compatible with the Le Fort I manoeuvre) were applied to complete the fracture. The bone interferences were removed, the maxilla easily positioned, and the procedure completed with the fixation of plates and screws from the 1.5-mm system (Tóride Indústria e Comércio Ltda., Mogi Mirim, São Paulo, Brazil).
The immediate postoperative period was uneventful and the patient was transferred from the post-anaesthetic recovery room to a standard occupancy room, completely awake and without any neurological symptoms. Eight hours after the surgical procedure, the patient presented one episode of nausea and vomiting, followed by lethargy. Unfortunately, the Oral and Maxillofacial Surgery (OMS) staff were not informed of these important events. Fifteen hours after the surgical procedure, the hospital nursing staff contacted the OMS staff, reporting that the patient presented left hemiplegia, hemiparaesthesia, and anisocoria. In addition to these signs, the patient was confused and complaining of severe headache and was promptly transferred to the intensive care unit (ICU) and the Neurosurgery staff were contacted. A cranial computed tomography (CT) scan was acquired, showing an ischemic area in the right middle cerebral artery region (MCA) without midline shift ( Fig. 1 A).
Angiography revealed a right internal carotid artery-to-jugular vein fistula at the level of the foramen lacerum, with high venous flow to the jugular bulb, superior and inferior petrous sinuses, cavernous sinus, and with reflux into the contralateral jugular vein ( Fig. 4 A,B).
After 3 days in the ICU, the patient was clinically stable, conscious, and maintaining left hemiplegia, hemiparaesthesia, and anisocoria. The patient complained of pulsating pain behind the right eye; however physical examination did not reveal chemosis, protosis, or a bruit at auscultation. Despite clinical stability, serial CT scanning revealed progressive enlargement of the hypodense area with midline shift ( Fig. 1 B). Owing to imminent herniation and risk of neurological decline, a decision was made to perform a decompressive craniectomy ( Fig. 1 C,D). A reverse question mark incision was made over the frontotemporoparietal region, extending down anterior to the tragus. A large hemicraniectomy was performed using a Midas Rex Legend drill (Medtronic Inc., Minneapolis, MN, USA) and additional bone was removed from the sphenoid wing, above the zygoma to decompress the basal cisterns. The dura was opened in a stellate fashion. To allow expansion of the oedematous brain tissue, the dura was left open and a large pericranium patch placed over it and anchored on five points. Finally, the galea layer and the skin were closed in a standardized fashion. One week after the craniectomy, the patient’s pupils were isochoric and he reported improvement of light touch sensibility on the left side of the body.
Twenty days after the craniectomy, the endovascular approach was planned as treatment for the fistula. Through an ipsilateral femoral approach, a vertebral 5F catheter was positioned in the left vertebral artery guided by the angiography. An Amplatz Super Stiff 0.035-mm Guidewire (Marlborough, Massachusetts, USA) was advanced beyond the fistula, followed by progression of the long inductor above the point of the fistula. The V12 6 × 38 mm stent was mounted on a balloon advanced over the 0.035-mm guide and positioned covering the carotid–jugular fistula. The stent was released under gradual balloon insufflation, aiming for the restitution of the usual calibre of the right internal carotid artery ( Fig. 4 C,D). After 30 days in hospital, the patient was discharged and sent for physical rehabilitation therapy.
At the 3-month follow-up, the patient showed significant improvement of hemiplegia and hemiparaesthesia, including being able to walk with the aid of crutches. A new CT scan revealed the stent position and a defect in the skull bones caused by decompressive craniectomy (Figs. 2 A–D and 4 C,D ). At the 36-month follow-up and after intensive physiotherapy, only moderate weakness and movement limitation in the left arm was present on the physical examination. The patient presented no difficulty in walking without aid and was able to drive without any car adaptations. It is important to note that the patient was extremely cooperative throughout the proposed treatment plan. The most recent CT scan shows the reconstruction of the bone defect caused by craniectomy with an acrylic prosthesis ( Fig. 3 E–H).
